Li et al.

BMC Neurology (2019) 19:43

https://doi.org/10.1186/s12883-019-1272-z

CASEREPORT Open Access

Isolated subdural hematoma secondary

to Dural arteriovenous fistula: a case
report and literature review
1† 2† 2 2 2* 2*
Guichen Li , Yang Zhang , Jinchuan Zhao , Xiaobo Zhu , Jinlu Yu and Kun Hou

Abstract
Background: Dural arteriovenous fistula (DAVF) is an uncommon subtype among the intracranial arteriovenous
malformations, which is characterized by pathological anastomoses between meningeal arteries and dural venous
sinuses, meningeal veins, or cortical veins. While intracerebral hemorrhage accounts for most of the hemorrhagic
cases in patients with DAVF, isolated subdural hematoma (SDH) is rarely reported.
Case presentation: A 45-year-old man was admitted for a progressively worsening headache over 2 weeks. Head
computed tomography on admission revealed an isodense chronic SDH (CSDH) on the left hemisphere with mild
midline shift. Further angiography of the external carotid artery revealed a DAVF at the transverse sinus. The DAVF
was embolized via the middle meningeal artery. His CSDH gradually resolved without surgical intervention. In order
to further elucidate this rare entity, a review of relevant literature was also conducted.
Conclusions: Isolated SDH is a rare complication of DAVF. In this report, we presented a rare case of
CSDH secondary to an intracranial DAVF. According to this case report and our literature review, the so-
called benign type of DAVF without cortical venous drainage does not always warrant a benign process
and might be complicated with SDH. Careful preoperative investigation is needed for relative young
patients presenting with idiopathic or atypical SDH.
Keywords: Dural arteriovenous fistula, Subdural hematoma, Cortical venous drainage, Middle meningeal artery

Background report a rare case of DAVF presenting with isolated

Dural arteriovenous fistula (DAVF) is an uncommon chronic subdural hematoma (CSDH). Furthermore, a
subtype among the intracranial arteriovenous malforma- review of the literature was also conducted to further
tions (AVMs), which is characterized by pathological illustrate the clinical profiles of this rare entity.
anastomoses between meningeal arteries and dural venous
sinuses, meningeal veins, or cortical veins [1]. From a Case presentation
recent epidemiologic survey of DAVF in Japan, the initial A 45-year-old man was admitted for a progressively
clinical presentation was intracranial hemorrhage in 16% worsening headache over 2 weeks. He denied history of
of the inflicted patients [2]. While intracerebral recent head trauma or anticoagulation and antiplatelet
hemorrhage (ICH) accounts for most of the hemorrhagic medication. General and neurologic examinations were
cases, isolated subdural hematoma (SDH) is rarely not remarkable on admission. Routine laboratory
reported [3, 4]. In the current study, we investigations including coagulation profiles and platelet
function were within normal limits. Head computed
tomography (CT) on admission revealed an isodense
* Correspondence: jlyu@jlu.edu.cn; houkunscience@163.com; CSDH on the right hemisphere with mild midline shift
houkunsciene@163.com
Guichen Li and Yang Zhang contribute equally to this manuscript and they
(Fig. 1a). A CT angiography (CTA) was performed to
are co-first authors. rule out any intracranial vascular malformation. A DAVF
2
Department of Neurosurgery, The First Hospital of Jilin University, was noticed at the transverse sinus with dilated cortical
71 Xinmin Avenue, Changchun, Jilin 130021, China
venous drainage (Fig. 1b). So, a digital subtraction
Full list of author information is available at the end of the article
© The Author(s). 2019 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the
Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Li et al. BMC Neurology (2019) 19:43
Fig. 1 (a) Head CT shows an isodense CSDH on the right hemisphere with mild midline shift. (b) CTA reveals a DAVF located at
the transverse sinus with dilated cortical venous drainage (arrow). CT: computed tomography; CSDH: chronic subdural
hematoma; CTA: CT angiography; DAVF: dural arteriovenous fistula
angiography (DSA) of the external carotid artery and
DAVF embolization was planned.
No anomaly was noticed during selective angiography
of the internal carotid and vertebral arteries and the left
external carotid artery. Selective angiography of the right
external carotid artery showed that the DAVF was lo-
cated at the transverse sinus and fed by posterior branch
of the middle meningeal artery (MMA), the occipital
artery, and the posterior meningeal artery and drained to
the occipital cortical veins with venous ectasia (Fig. 2a-b).
The DAVF was classified as type IV accord-ing to the
Cognard classification. The embolization was performed
via the MMA. The Headway duo catheter was used and
accessed to the DAVF, and Onyx was injected until the
shunt disappeared (Fig. 2c-d). The patient experienced an
uneventful recovery. His CSDH gradually resolved in 1
month (Fig. 3). No neurologic deficit was noticed.
Literature review
A PubMed search of published studies written in English
and Chinese was conducted on June 30th, 2017. The
following key words were used in relevant combinations:
dural arteriovenous fistula, dural arteriovenous
malformation, subdural hematoma, subdural haema-toma,
subdural hemorrhage, and subdural haemorrhage. The
reference lists of the identified articles were also manually
searched for additional studies. Studies of which full text
could not be obtained or those without sufficient
individualized description of the isolated SDH cases
mixed in larger case series were excluded.
Finally, 13 articles containing 14 patients were identi-
fied [3–15]. In all 15 patients (9 females, 60%) including
1 case in our institution were included for the final in-
terpretation (Table 1). The inflicted patients were aged
from 27 to 82 years (55.5 ± 8.6).Of note, 12 (80%) of the
15 patients were aged between 40 and 60 years of age,
Page 2 of 6
and 8 (53.3%) patients were between 50 and 60 years.
Sides of the DAVF or SDH were obtained in 13 patients
with 9 (69.2%) located at the left side and 4 at the right
side. The intracranial locations of DAVF were anterior
fossa (2), middle fossa (2), frontal region (2), parietal
region (3), temporal region (2), and occipital region (2).
The types of SDH were CSDH (7/15), acute SDH
(ASDH) (7/15), and undefined SDH (1/15). Of the 12
patients feeding artery could be identified, MMA was the
commonest feeding artery (8/12, 66.7%). Multiple
feeding arteries were identified in 3 (25%) patients. The
causes of DAVF were only defined in 3 patients (2 iatro-
genic and 1 traumatic), with the rest undefined or not
mentioned. Cognard classifications (Table 2) of the
DAVF were reported or deduced from the reports in 12
patients, with type I, type III, and type IV in 7, 2, and 3
patients respectively. The treatment strategies included
hematoma evacuation (2/15), CSDH drainage and DAVF
embolization (5/15), craniotomy and DAVF re-section
(3/15), DAVF embolization and hematoma evacuation
(1/15), DAVF embolization (2/15), and not applicable or
not mentioned (2/15). Of the 12 pa-tients with direct
description of outcome, 7 (58.3%) patients were
neurological intact, 3 (25%) patients with neurological
deficits, and 2 (16.7%) died.
Discussion
DAVF is an uncommon subtype of intracranial AVMs
[1]. In a Scottish population-based study in adults, the
detection rate of DAVF was 0.16 per 100,000 adults per
year, whereas the rate of all intracranial vascular
malformations was 2.27 per 100,000 adults per year in the
same population [16]. The manifestations of DAVF are
diverse. In the recent Japanese survey by Kuwayama N et
al., the initial clinical presentation was ocular symptoms,
tinnitus, intracranial hemorrhage, and non-hemorrhagic
neurological deficits in 45, 20, 16, and
Li et al. BMC Neurology (2019) 19:43 Page 3 of 6

Fig. 2 (a) Preoperative DSA of the right external carotid artery shows that the DAVF is fed by the branch of MMA and OA, and drains to
the dilated occipital cortical vein. (b) DSA of the right vertebral artery shows that the DAVF also receives blood supply from the PMA. (c-
d) DAVF disappears after Onyx embolization injection via the branch of MMA. DSA: digital subtraction angiography; DAVF: dural
arteriovenous fistula; MMA: middle meningeal artery; OA: occipital artery; PMA: posterior meningeal artery

20% of the patients respectively [2]. In the hemorrhagic

patients, isolated SDH was only reported sporadically [3–
15]. According to the literature, the majority of DAVFs
are acquired in an idiopathic fashion, only a small
proportion results from causes as trauma, infection, and
iatrogenic injury [1, 17]. In this study, in-cluding our
case, the causes of DAVF and associated SDH were only
defined in 3 patients (2 iatrogenic and 1 traumatic), with
the rest undefined or not mentioned.
The initial clinical presentation is not specific in
patients with DAVF associated SDH. Just as presented in
our case, headache (chronic, acute, or progressive) was
the most common complaint. As a result of its rarity in
occurrence, hardly could we ever associate DAVF with an
SDH. In case of a patient presenting with SDH, there is
no specific indication in imaging and clinical presenta-
tion that could imply the possibility of an underlying
DAVF. However, there are some points that might indi-
cate the existence of some underlying disorders: a) rela-
tively young age, b) no evident history of head trauma,
c) no coagulopathy or anticoagulation and antiplatelet
medication, d) spontaneous occurrence of SDH, e) re-
Fig. 3 Head CT shows spontaneous resolution of the CSDH. CT:
current or refractory SDH that recurs in a short period
computed tomography; CSDH: chronic subdural hematoma
after previous satisfactory hematoma evacuation.
 Li et al. BMC Neurology
Table 1 Clinical data of the patients with DAVF associated isolated SDH
Patient Location of Feedin Cause Type
Reference (Age, DAVF g of of Cognard Treatment Outcome
classifica
Sex) artery DAVF SDH tion
64 Midline of the Undefi ASD Hematoma
Ito et al., 1983 [5] years, M anterior OPA ned H NA/NM evacuation with NA/NM
fossa (R) DAVF untreated

(2019) 19:43
Halbach et al., 48 Bilater Undefi CSD CSDH drainage Complete
1988 [6] years, F Parietal (R) al ned H NA/NM and direct resolution
MMA puncture
MMAs and
embolization
Pappas CT et al., 58 Iatroge CSD Craniotomy and Mild expressive
1992 [7] years, F Parietal (L) MMA nic H Type I DAVF aphasia
resection
Başkaya MK et al., 51 Anterior fossa Undefi ASD Craniotomy and Without
1994 [8] years, F (L) AEA ned H Type I DAVF Neurologic deficit
resection
Komiyama M et al., 58 CSD DAVF Without
1994 [9] years, F Temporal (L) MMA Head H Type I embolization and Neurologic deficit
Traum burr-hole
a drainage
Duffau H et al., 55 NA/N DAVF
1999 [10] years, M NA/NM M NA/NM SDH Type III embolization and Improved
Hematoma
evacuation
56 NA/N ASD Hematoma
years, F Middle Fossa M NA/NM H Type III evacuation Death
Maiuri F et al., 59 Undefi CSD
2001 [3] years, F Occipital (L) MMA ned H TypeIV NA/NM NA/NM
Kominato et al., 42 NA/N Undefi ASD
2004 [11] years, F NA/NM (L) M ned H NA/NM NA/NM Death
Kohyama S et al., 60 Middle Fossa Bilater Undefi ASD DAVF No neurological
2009 [12] years, M (L) al ned H TypeI embolization and deficit
burr-hole
MMAs drainage
Ogawa K et al., 27 Undefi ASD Hematoma No neurological
2010 [13] years, M Parietal (L) OA ned H Type I evacuation and deficit
DAVF resection
de Aguiar GB et al., 60 Undefi ASD DAVF
2016 [14] years, F Frontal (R) STA ned H TypeIV embolization Improved
Mewada T et al., 82 Iatroge CSD Burr-hole drainage
2016 [15] years, F Frontal (L) MMA nic H Type I and DAVF NA/NM
embolization
Kim E et al., 2016 67 Undefi CSD Burr-hole drainage Complete

Page 4 of
[4] years, M Temporal (L) MMA ned H Type I and DAVF resolution
embolization
45 MMA, Undefi CSD DAVF Complete
Present case years, M Occipital (R) PMA, ned H TypeIV embolization resolution
OA
M male, F: female, R right, L left, NA/NM not applicable or not mentioned, DAVF dural arteriovenous fistula, MMA middle meningeal
artery, OPA ophthalmic artery, OA occipital artery, PMA posterior meningeal artery, AEA anterior ethmoidal artery, STA superficial
temporal artery, SDH subdural hematoma, ASDH acute subdural hematoma, CSDH chronic subdural hematoma
Li et al. BMC Neurology (2019) 19:43
Table 2 Cognard classification of intracranial DAVF
Type Venous drainage
Type I Anterograde drainage into venous sinus
Type II
IIA Venous drainage into dural sinus with retrograde flow
IIB Venous drainage into dural sinus with normal
antegrade flow and CVD
IIA + B Venous drainage into dural sinus with retrograde
flow and CVD
Type III Venous drainage directly into subarachnoid vein (CVD only)
Type IV Venous drainage directly into subarachnoid vein with
venous ectasia
Type V Venous drainage directly into spinal perimedullar veins
CVD cortical venous drainage
The natural history of DAVF is primarily determined by
the pattern of venous drainage [1, 17, 18]. Patients with
cortical venous drainage (CVD) (especially with venous
ectasia) have an aggressive natural history including ICH
and nonhemorrhagic neurologic deficits (NHNDs). While
DAVF without CVD manifests a benign process and
rarely causes ICH or NHNDs. In this literature review,
Cognard classification (Table 2) of the DAVF were
reported or deduced from the reports in 12 patients, with
type I, type III, and type IV in 7, 2, and 3 patients,
respectively. Seven (58.3%) of the 12 patients harbored
the supposed benign type I DAVF, which is somewhat
inconsistent with the classical viewpoint [1, 17]. Hence,
DAVF with CVD demonstrates an aggressive natural
history and might be complicated with any kind of
intracranial hemorrhage including SDH. The so-called
benign type of DAVF without CVD does not always war-
rant a benign process and could also be complicated with
SDH.
There was no consensus on the treatment of DAVF and
its associated SDH. The treatment strategies depend on
specific circumstances. In the case of massive ASDH,
hematoma evacuation combined with simultaneous
DAVF resection was the preferred strategy [7, 8, 13].
While burr-hole drainage combined with DAVF
embolization was more suitable for CSDH patients [4, 6,
7, 12]. When the SDH did not cause evident intracranial
hypertension or neurological deficit, mere DAVF
embolization could also be selected [4, 14, 15]. Because
the mass effect of CSDH in our patient was mild, we just
embolized the DAVF at primary treatment. And the
CSDH resolved spontaneously.
Limitations
This is an isolated case report and a review of the
literature. The data we interpreted were extracted from
sporadic cases. As a result of the nature of this study,
statistical analysis could not be conducted. Hence, the
Page 5 of 6
conclusion we achieved in the text is just a narrative
interpretation of the past studies and future studies with
larger case series are anticipated.
Conclusion
DAVF is an uncommon subtype of intracranial AVMs.
Isolated SDH including ASDH and CSDH is a rare
complication of DAVF. The so-called benign type of
DAVF without CVD does not always warrant a benign
process and could also be complicated with SDH. Careful
preoperative investigation is needed for relative young
patients presenting with idiopathic or atypical SDH.
Abbreviations
ASDH: Acute subdural hematoma; AVMs: Arteriovenous malformations;
CSDH: Chronic subdural hematoma; CT: Computed tomography; CTA: CT
angiography; CVD: Cortical venous drainage; DAVF: Dural arteriovenous
fistula; DSA: Digital subtraction angiography; ICH: Intracerebral hemorrhage;
MMA: Middle meningeal artery; NHNDs: Nonhemorrhagic neurologic deficits;
SDH: Subdural hematoma
Acknowledgements
None.
Funding
None.
Availability of data and materials
The datasets used and analysed during the current study are
available from the corresponding author on reasonable request.
Authors’ contributions
Conception and design: GL,XZ, YZ. Acquisition of data: JZ, JL. Analysis
andinterpretation of data: KH, GL. Drafting the article: GL, KH.
Critically revising the article: JL, KH. All of the authors have read
and approved the final manuscript.
Ethics approval and consent to participate
This study was approved by the institutional review board of The
First Hospital of Jilin University and informed written consent was
obtained from the patient.
Consent for publication
Written informed consent was obtained from the patient for publication of
this manuscript and any accompanying images. Copy of the written
consent is available for review by the editor of this journal.
Competing interests
The authors declare that they have no competing interests.
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional
claims in published maps and institutional affiliations.
Author details
1
Department of Neurology, The First Hospital of Jilin University,
2
Changchun, Jilin, China. Department of Neurosurgery, The First Hospital
of Jilin University, 71 Xinmin Avenue, Changchun, Jilin 130021, China.
Li et al. BMC Neurology (2019) 19:43 Page 6 of 6

Received: 16 November 2017 Accepted: 12 March 2019

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